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Diabetic Muscle Infarction: a Rare Cause of Acute Limb Pain in Dialysis Patients Hindawi Publishing Corporation Case Reports in Nephrology Volume 2013, Article ID 931523, 6 pages http://dx.doi.org/10.1155/2013/931523 Case Report Diabetic Muscle Infarction: A Rare Cause of Acute Limb Pain in Dialysis Patients G. De Vlieger,1 B. Bammens,1,2 F. Claus,3 R. Vos,4 and K. Claes1,2 1 Department of Nephrology and Renal Transplantation, University Hospitals Leuven, Herestraat 49, 3000 Leuven, Belgium 2 Laboratory of Nephrology, Department of Microbiology and Imunology, KU Leuven, Herestraat 49, 3000 Leuven, Belgium 3 Department of Radiology, University Hospitals Leuven, Herestraat 49, 3000 Leuven, Belgium 4 Respiratory Division, University of Leuven and Department of Clinical Respiratory Medicine, Catholic University of Leuven, Herestraat 49, 3000 Leuven, Belgium Correspondence should be addressed to G. De Vlieger; [email protected] Received 18 March 2013; Accepted 7 April 2013 Academic Editors: J. Almirall, P. S. Passadakis, and H. Schiffl Copyright © 2013 G. De Vlieger et al. This is an open access article distributed under the Creative Commons Attribution License, which permits unrestricted use, distribution, and reproduction in any medium, provided the original work is properly cited. Diabetic muscle infarction is a rare microangiopathic complication occurring in patients with advanced diabetes mellitus. Diabetic patients with chronic kidney disease stage Vd are prone to develop this complication. The presenting symptom is a localized painful swelling of the affected limb. Symptoms usually resolve spontaneously during the following weeks, but frequent relapse can occur and in some cases swelling may lead to compartment syndrome. Biochemical blood analyses show an elevated C-reactive protein, but creatine kinase is often normal. Diagnosis can be made on clinical presentation and imaging, with magnetic resonance imaging as the gold standard. Histology is often not contributive. Treatment consists of rest, analgesics, rigorous glycemic control and low- dose aspirin. Severe cases of compartment syndrome require fasciotomy. In the current paper, we present two diabetic patients with cystic fibrosis, who are treated with automated peritoneal dialysis and suffered from episodic lower limb infarction. Wesubsequently review 48 episodes of diabetic muscle infarction previously reported in the literature in patients with end-stage renal disease. 1. Introduction (PD) was started. Two years later, she presented with acute pain in the right calf. Biochemical evaluation showed an Diabetic muscle infarction (DMInf) is a rare microangio- elevated creatine kinase (CK 218 U/L) and C-reactive protein pathic complication in patients with advanced diabetes melli- (CRP 97 mg/L). HbA1c was 5.8%. Ultrasound and computed tus (DM). Patients having terminal diabetic nephropathy are tomography (CT) showed diffuse muscular and subcuta- prone to develop DMInf and nearly one-fourth of DMInf, neous edema of the affected calf. Muscular biopsy demon- patients receive renal replacement treatment [1]. Conse- strated muscular atrophy, macrophages, and myophagia. The quently, nephrologists are likely to be increasingly confronted symptoms resolved within four weeks. There was a new onset with this disease entity. of pain in the left calf 18 months later. CK was normal, but CRP levels were elevated (215 mg/L). HbA1c was 7.2%. The 2. Case Reports clinical and biochemic characteristics are shown in Table 1. Magnetic resonance imaging (MRI) showed an infarction Case 1. A 27-year-old woman with cystic fibrosis started in the soleus muscle (Figures 1(a) and 1(b)). During the insulin treatment at the age of 11. When she was 16 years following days, pain increased and a compartment syndrome old, she received bilateral lung transplantation (SSLTx). Her was diagnosed by pressure measurement. A fasciotomy was immune-suppressive therapy consisted of tacrolimus and performed, and low-dose aspirin was started with subse- steroids. At the age of 24, she developed chronic kidney quent resolution of the complaints in the following two disease stage V (CKD-Vd) for which peritoneal dialysis months. 2 Case Reports in Nephrology (c) (a) (b) (d) Figure 1: T2 weighted images with coronal (a, c) and axial (b, d) views for the 2 patients. The level of the axial views are indicated onthe coronal images by the dashed lines. Images (a) and (b) show diffuse edema in the calf muscles of the left lower limb of the first patient, as illustrated by the hyperintense signal of the muscular tissue (note the normal low signal intensity of the muscles in the right leg). Images (c) and (d) show similar changes in the adductor muscles of the left upper leg of the second patient. Case 2. A 35-years-old woman with cystic fibrosis received 3. Discussion SSLTx at the age of 21. Redo SSLTx because of chronic lung allograftrejectionwasperformedsixyearslater.Immuno- DMInf was first described in 1965 by Angervall and Stener suppressive treatment consisted of tacrolimus and steroids. [2]. It is a rare microangiopathic complication in patients with DM was diagnosed at the age of 29, and subcutaneous advanced DM. The typical presentation is an acute onset of insulin treatment was initiated one year later. When she severe pain at rest, exacerbated by movement. Clinical exam- ination usually reveals a hard and warm palpable swelling of was 34 years old, PD was started. Six months later, she was the affected limb, and ultrasound is negative for DVT. admitted because of pain in the left thigh. The pain was In a review of 166 episodes in 115 diabetic patients by present during rest and increased during exercise. Clinical Trujillo-Santos, the mean age at presentation of DMInf was examination revealed a swollen and painful left upper leg with 42.6 years, and the mean duration of DM was 14.3 years absence of deep venous thrombosis (DVT) on ultrasound. with 59% of the patients suffering from DM type17 1[ ]. MRIshowedmuscleinfarctionwithcentralnecrosisin A female predominance (61,5%) was observed, and associ- theadductors,medialvastusmuscle,andsartoriusmuscle ated microangiopathic complications included nephropathy, (Figures 1(c) and 1(d)). CRP was elevated (94 mg/L), but retinopathy, and neuropathy in 71.1, 56.6, and 54.5%, respec- CK’s were repeatedly normal. The DM was poorly controlled tively [17]. We described two patients with cystic fibrosis with an HbA1c of 8.7%. Low-dose aspirin was started, and who evolved to CKD-V after SSLTx. Both patients were thesymptomsresolvedaftersixweeks.Sixmonthslater, treated with PD and were at high risk for the development shewasreadmittedbecauseofseverepainintheright of DMInf due to multiple risk factors: DM type 1, admin- thigh. Biochemical examination showed an increased CRP of istration of calcineurin inhibitors and prothrombotic status 104 mg/L, the CK was within the normal range, and HbA1c due to uremia. Although there is no evidence of increased was 7,5%. MRI showed edema in the left pectineus muscle, thrombogenic risk in cystic fibrosis, lung transplantation external obturator, and adductor muscles. Analgesia was is clearly associated with a hypercoagulability [18]andan started with spontaneous resolution. increased risk for DVT and pulmonary embolism [19]. Case Reports in Nephrology Table 1: Overview of reviewed cases in the literature. DM complications Laboratory Ref Age Gender Type DM Age at onset RRT Affected muscles other than CKD HbA1c CRP (mg/dL) CK (U/L) (1) Left vastus lateralis 24 Female DM I 11 Retinopathy APD 5.8 97 218 (2) Left calf [3]29FemaleDMI 9 Neuropathy SPK 5 — 160 Right gastrocnemius [4]29FemaleDMI 8 Retinopathy, neuropathy SPK — 17 Right gastrocnemius and soleus [5]31MaleDMI — Retinopathy, coronaropathy IHD — — — Left vastus lateralis (1) Right vastus lateralis Retinopathy, peripheral [6]31FemaleDMI 16 IHD 12.4 — 649 (2) Left thigh vascular disease (3) Left calf Retinopathy, neuropathy, and [7] 32 Male DM I 20 IHD — — 350 Left vastus muscles peripheral vascular disease Retinopathy, neuropathy, and [4]39FemaleDMI 9 SPK 5.0 — 21 Right deltoid muscle ischemic cardiac disease [4]33FemaleDMI 7 Retinopathy, neuropathy SPK 5.6 — Normal Left biceps brachii (1) Left vastus medialis and 35 Female DM I 30 APD 8.7 94.2 97 intermedius (2) Right adductors [4]39FemaleDMI 9 Retinopathy, neuropathy SPK, IHD 5.3 — 83 Right thigh adductors (1) Left thigh (2) Right vastus medialis [8]49MaleDMI — Retinopathy, neuropathy CAPD, IDH 7.9 214 — (3) Left thigh (4) Right calf (1) Left buttock [8]51MaleDMI 41 — IHD — 216 381 (2) Right semitendinosus [9]35MaleDMII 27 Retinopathy, coronary disease CAPD — — 556 Right vastus medialis [5]39MaleDMII 39 Retinopathy, neuropathy IHD — — Elevated Left gastrocnemius Retinopathy, neuropathy, and [8]40MaleDMII 25 CAPD, IHD — — — Left rectus femoris peripheral vascular disease (1) Right thigh [7]44FemaleDMII 41 Retinopathy, neuropathy IHD — — — (2) Left calf (1) Left calf [9]49FemaleDMII 34 Retinopathy, neuropathy CAPD, IHD — — Normal (2) Left gluteal region (3) Right thigh [10]49MaleDMII — Heart failure IHD — 35 289 Right rectus femoris [8]49FemaleDMII — Neuropathy IHD — 300 692 Anterior right thigh [11]51MaleDMII — — IHD — — Normal Left thigh [12]55MaleDMII 35 — IHD — — 463 Left vastus lateralis (1) Left thigh [13]55MaleDMII 31 Retinopathy, neuropathy CAPD — 105 Normal (2) Right thigh (3) Right thigh [5]56FemaleDMII — Retinopathy, neuropathy CAPD — — Elevated Left quadriceps Retinopathy, neuropathy, and [5]58MaleDMII — IHD — 70 Normal Bilateral quadriceps peripheral vascular disease 3 4 Table 1: Continued. DM complications
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